Radiolucency in the mandible: a seventeen-year follow-up Saydé Sokhn, CD, DUA, DUB, a Ibrahim Nasseh, DCD, DSO, FICD, b and Fawzi Karam, DCD, CES c School of Dentistry, Lebanese University, Beirut, Lebanon (Oral Surg Oral Med Oral Pathol Oral Radiol 2014;117:153-158) CLINICAL PRESENTATION In 2009, a 43-year-old white woman was referred to the Department of Oral and Maxillofacial Imaging at the Lebanese University with an approximately 1 year history of a slowly expanding, painless mass along the left inferior part of the mandible in the premolar region. The patient initially consulted her dentist in 2007, and a panoramic radiograph was acquired at that time (Figure 1). The practitioner told her that there is nothing to worry about, and he scheduled a follow-up appointment. Sixteen months later, the patient went to the dental school for a second opinion. She was referred to our department, where a panoramic radiograph was taken (Figure 2). It showed a well-circumscribed, round, radiolucent area extending from the inferior border of the mandible in the premolar region and overlapping the mandibular canal. Cone beam computed tomography imaging (using i-CAT (Imaging Sciences International, USA) with voxel size 0.3 mm (field of volume 6 cm), tube voltage of 120 kV, current of 23.87 mA, and exposure time of 20 s) demonstrated a 13 11 mm lytic lesion in the left mandibular premolar region. The lesion was situated on the buccal aspect in the inferior third of the mandible. The lesion was irregular in shape, noncorticated, but fairly well demarcated. The alveolar process was interrupted, with the presence of what seemed to be a communication between the buccal and lingual aspect and a bone structure connecting the mesial and distal sides. The mandibular canal was displaced to the lingual side; the lingual cortex was intact (Figure 3). During the clinical examination, the patient com- plained of mild pain over the swollen area; however, she reported that chewing, swallowing, and speech were normal. On clinical examination, there was a slightly firm swelling in the region, slightly painful on pressure. Skin and the oral mucosa over the swelling were normal; however, the swelling could not be palpated intra-orally. Dental implants were clinically and radiographically normal. The patient’s medical history was contributory: she revealed that an ossifying fibroma was resected from the same location 15 years ago. Her records revealed that, in 1995, the patient presented with a 2.5 2 cm painless, firm swelling in the same region; the adjacent teeth were vital and painless but mobile. The lesion was removed under locoregional anesthesia. A dissection of the mental nerve was undertaken to create a sufficient operating field. Severe paresthesia persisted for 3 months after the surgery. After all this time, we could not retrieve the glass slide or paraffin block from the previous biopsy to confirm or rule out the previous diagnosis of ossifying fibroma. One year after that surgery, a well-defined radiolu- cency was noted on the panoramic radiograph, and a depression was detected on clinical examination. This was determined to represent fibrous healing because of the large areas of interruption of the periosteum. An onlay bone graft from the chin was placed in the middle of the defect. One year after the bone graft, 2 dental implants were placed. Twelve years later, the patient returned, complaining of the same signs and symptoms. On January 2009, a surgery was performed under locoregional anesthesia. First, the mental nerve was carefully dissected to avoid perioperative damage. The lesion showed a very slight adherence to the surrounding bone and was easily removed in several pieces. The specimen was grayish in color, had a firm-elastic and connective tissue consis- tency, and measured 1 0.8 0.6 cm. The bone cavity was thoroughly and carefully curetted, taking care to not damage the mental nerve. DIFFERENTIAL DIAGNOSIS The differential diagnosis criteria were based on the patient’s age, gender, and clinical history, as well as on all the features of the lesion: location, boundary destruction, calcification, and the diagnosis of the lesion previously seen in the same area. The patient presented with a history of swelling of 2 years’ duration along the left inferior part of the mandible. Swelling was not associated with pain or paresthesia. Radiographically, multiple features needed to be addressed to establish the differential diagnosis. The interrupted alveolar process and the bone bridge, in the superior two-thirds of the alveolar process, are related to the history of previous a Clinical Instructor, Department of Oral and Maxillofacial Imaging, School of Dentistry, Lebanese University. b Chairperson, Department of Oral and Maxillofacial Imaging, School of Dentistry, Lebanese University. c Department of Oral and Maxillofacial Surgery, School of Dentistry, Lebanese University. Received for publication Jan 17, 2013; returned for revision May 20, 2013; accepted for publication Jun 2, 2013. Ó 2014 Elsevier Inc. All rights reserved. 2212-4403/$ - see front matter http://dx.doi.org/10.1016/j.oooo.2013.06.001 153 Vol. 117 No. 2 February 2014
Transcript
Vol. 117 No. 2 February 2014
Radiolucency in the mandible: a seventeen-year follow-upSaydé Sokhn, CD, DUA, DUB,a Ibrahim Nasseh, DCD, DSO, FICD,b and Fawzi Karam, DCD, CESc
School of Dentistry, Lebanese University, Beirut, Lebanon(Oral Surg Oral Med Oral Pathol Oral Radiol 2014;117:153-158)
CLINICAL PRESENTATIONIn 2009, a 43-year-old white woman was referred to theDepartment of Oral and Maxillofacial Imaging at theLebanese University with an approximately 1 yearhistory of a slowly expanding, painless mass along theleft inferior part of the mandible in the premolar region.The patient initially consulted her dentist in 2007, anda panoramic radiograph was acquired at that time(Figure 1). The practitioner told her that there is nothingto worry about, and he scheduled a follow-upappointment. Sixteen months later, the patient went tothe dental school for a second opinion. She was referredto our department, where a panoramic radiograph wastaken (Figure 2). It showed a well-circumscribed,round, radiolucent area extending from the inferiorborder of the mandible in the premolar region andoverlapping the mandibular canal.
Cone beam computed tomography imaging (usingi-CAT (Imaging Sciences International, USA) withvoxel size 0.3 mm (field of volume 6 cm), tube voltageof 120 kV, current of 23.87 mA, and exposure time of20 s) demonstrated a 13 � 11 mm lytic lesion in the leftmandibular premolar region. The lesion was situated onthe buccal aspect in the inferior third of the mandible.The lesion was irregular in shape, noncorticated, butfairly well demarcated. The alveolar process wasinterrupted, with the presence of what seemed to bea communication between the buccal and lingual aspectand a bone structure connecting the mesial and distalsides. The mandibular canal was displaced to thelingual side; the lingual cortex was intact (Figure 3).
During the clinical examination, the patient com-plained of mild pain over the swollen area; however,she reported that chewing, swallowing, and speechwere normal. On clinical examination, there wasa slightly firm swelling in the region, slightly painful onpressure. Skin and the oral mucosa over the swellingwere normal; however, the swelling could not be
aClinical Instructor, Department of Oral and Maxillofacial Imaging,School of Dentistry, Lebanese University.bChairperson, Department of Oral and Maxillofacial Imaging, Schoolof Dentistry, Lebanese University.cDepartment of Oral and Maxillofacial Surgery, School of Dentistry,Lebanese University.Received for publication Jan 17, 2013; returned for revision May 20,2013; accepted for publication Jun 2, 2013.� 2014 Elsevier Inc. All rights reserved.2212-4403/$ - see front matterhttp://dx.doi.org/10.1016/j.oooo.2013.06.001
palpated intra-orally. Dental implants were clinicallyand radiographically normal.
The patient’s medical history was contributory: sherevealed that an ossifying fibroma was resected fromthe same location 15 years ago. Her records revealedthat, in 1995, the patient presented with a 2.5 � 2 cmpainless, firm swelling in the same region; the adjacentteeth were vital and painless but mobile. The lesion wasremoved under locoregional anesthesia. A dissection ofthe mental nerve was undertaken to create a sufficientoperating field. Severe paresthesia persisted for 3months after the surgery. After all this time, we couldnot retrieve the glass slide or paraffin block from theprevious biopsy to confirm or rule out the previousdiagnosis of ossifying fibroma.
One year after that surgery, a well-defined radiolu-cency was noted on the panoramic radiograph, anda depression was detected on clinical examination. Thiswas determined to represent fibrous healing because ofthe large areas of interruption of the periosteum. Anonlay bone graft from the chin was placed in the middleof the defect. One year after the bone graft, 2 dentalimplants were placed.
Twelve years later, the patient returned, complainingof the same signs and symptoms. On January 2009,a surgery was performed under locoregional anesthesia.First, the mental nerve was carefully dissected to avoidperioperative damage. The lesion showed a very slightadherence to the surrounding bone and was easilyremoved in several pieces. The specimen was grayish incolor, had a firm-elastic and connective tissue consis-tency, and measured 1 � 0.8 � 0.6 cm. The bone cavitywas thoroughly and carefully curetted, taking care tonot damage the mental nerve.
DIFFERENTIAL DIAGNOSISThe differential diagnosis criteria were based on thepatient’s age, gender, and clinical history, as well as onall the features of the lesion: location, boundarydestruction, calcification, and the diagnosis of the lesionpreviously seen in the same area. The patient presentedwith a history of swelling of 2 years’ duration along theleft inferior part of the mandible. Swelling was notassociated with pain or paresthesia. Radiographically,multiple features needed to be addressed to establish thedifferential diagnosis. The interrupted alveolar processand the bone bridge, in the superior two-thirds of thealveolar process, are related to the history of previous
Fig. 1. Panoramic radiograph acquired 2 years ago, showing a radiolucency within the inferior border of the left mandible (arrows).
Fig. 2. The present panoramic radiograph, showing more clarity, depicting a radiolucent process at the inferior border of the leftside of the mandible (arrows).
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surgery. This area is not affected by the current lesion,because of the smooth borders and the absence of anylesional tissue. Our attention was focused on the lesionnoted in the buccal part of the inferior third of themandible.
Our differential diagnosis included recurrence of theprevious ossifying fibroma, traumatic bone cavity(TBC)/cyst, aneurysmal bone cyst (ABC), central giantcell lesions, intraosseous carcinoma, and desmoplasticfibroma (DF), with an unlikely possibility of a traumaticneuroma.
Ossifying fibromas arise across a wide age range,with the highest number of cases noted during the thirdand fourth decades with female predilection. Themandible is more affected than the maxilla, especiallyin the premolar and molar regions. Radiographically,the lesion most often is well defined, which was thecase in the current lesion; however, the lesion iscompletely radiolucent, which is not the mostfrequently seen presentation of ossifying fibroma.Depending on the amount of calcified materialproduced in the tumor, in more than two-thirds of the
Fig. 3. Cone beam computed tomography (panoramic reconstruction with cross-section images) showing the expansion of thebuccal cortical bone and the true dimensions of the lesion.
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cases, it presents with varying degrees of radiopacity,going from some radiopaque foci, to a central opacifi-cation, and to ground glass or mottled radiopacities.The absence of internal opacity reduces the possibilityof a recurrent ossifying fibroma.
TBC is unilocular and radiolucent, affecting themandible more than the maxilla; large, expansile, and
multilocular TBCs have been described. In this case,a surgery was done with large interruption of the peri-osteum that might cause a local ischemic accidentleading to a solitary (traumatic) bone cyst. However,TBCs are asymptomatic in the majority of cases, andare often accidentally discovered on routine radiologicexamination. They frequently present with a scalloped
Fig. 4. Histopathologic section showing a spindle cellproliferation arranged in diffuse fascicles (hematoxylin-eosin(H&E) stain, original magnification �100).
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superior border, spreading between the roots of adjacentvital teeth. Expansion is not characteristic of traumaticbone cysts, but it is described in about 26% of the cases.
ABC is a benign cystic lesion of bone, composed ofblood-filled spaces separated by connective tissue septacontaining fibroblasts, osteoclast-type giant cells, andreactive woven bone. Fifty percent of ABCs arise in thelong bones and 20% in the vertebral column. Itaccounts for 1.5% of the nonodontogenic, nonepithelialcysts of the mandible. It is found more frequently in themandible than in the maxilla (3:1) with preponderancefor the body, ramus, and angle of the mandible. Morethan 90% of reported jaw lesions have occurred inindividuals younger than 30 years of age, with a predi-lection for females. The etiology of ABC is con-troversial. It can develop as either a primary ora secondary lesion associated with other bone diseases.A history of trauma and subperiosteal hematomaformation is an essential factor in the development ofABC.1 An ABC in the jaw usually manifests as a fairlyrapidly developing bony swelling (usually buccal orlabial). Pain is an occasional complaint, and theinvolved area may be tender on palpation.2,3 ABCs areseen in young females, under 20 years of age, and tendto have an eccentric, “blown-out” appearance. In ourcase, the first lesion occurred when the patient was inher late 30s, and the shape of her lesion was morefusiform and confined to the mandible boundaries, withlimited expansion.
Central giant cell granuloma (CGCG) of the jawaffects the mandible in about two-thirds of cases, withmost tumors being located anterior to the second molar;females are affected more than males. The behavior ofCGCG is variable, most commonly producing anasymptomatic expansion of the jaws. However, it canbe clinically aggressive, associated with pain, osseousdestruction, and cortical perforation. Around 60% ofCGCG cases occur in individuals younger than 20years; the lesion frequently crosses the midline andoften presents with thin internal septations. The shapeand location of the lesion and the absence of thininternal septa, as well as the age of the patient when thefirst lesion occurred, slightly reduce the possibility ofCGCG in this patient.
Primary intraosseous carcinoma of the mandible israre and may remain silent until it has reached a fairlylarge size. Pain, pathologic fracture, trismus, lymph-adenopathy, and sensory nerve abnormalities, such aslip paresthesia, may occur with this tumor. It is morecommon in men and in patients in their fourth to ninthdecades of life. These factors place this diagnosis lowon the differential diagnosis.
The differential diagnosis can also include traumaticneuroma. The manipulation of the mental nerve in 1995and the long-standing postoperative paresthesia suggest
some nerve damage. Traumatic neuromas appearusually a long time after a surgery performed close toa nerve emergence and when the nerve was dissected orintentionally displaced. Radiographically, the lesion isradiolucent and uniform but should appear to bedirectly related to a foramen, unlike the present case.
Differential diagnosis also includes a central fibroma,which can involve the jaw. Depending on the presence orabsence of odontogenic epithelia, intraosseous fibromacases can be classified into 2 respective categories:odontogenic fibroma or nonodontogenic fibroma (des-moplastic fibroma [DF]). The clinical findings forintraosseous odontogenic fibroma are similar to thoseseen with DF in the jaws. However, DF is generallybelieved to be more aggressive than odontogenicfibroma.2-4 In general, the radiographic features of DFare nonspecific. These include a unilocular or multi-locular, well-demarcated or irregular radiolucency withvariably expressed marginal sclerosis.5-8 The currentlesion, at the very least, could also represent the recur-rence of a previously misdiagnosed lesion, which wasincompletely excised.
DIAGNOSISHistopathologic examination revealed a moderatelycellular fibrous connective tissue richly vascularized andfocally hyalinized, composed of spindle-shaped cellsarranged in dispersed fascicles (Figure 4) and abundantkeloid-like collagen formation (Figure 5). Some residualbone trabeculae are seen at the periphery (Figure 6).There is also a lack of cytologic features of malignancy(Figure 7). No epithelial islands are seen. On the basisof these characteristics, the diagnosis of a DF was made.
In general, the radiographic features of DF arenonspecific. These include a unilocular or multilocular,well-demarcated or irregular radiolucency with variably
Fig. 5. Abundant dense matrix of keloid-like collagenformation (H&E, �400).
Fig. 6. Some residual bone trabeculae existing at theperiphery (H&E �40).
Fig. 7. Bland cytologic histopathologic features, suggestingabsence of malignancy (H&E �200).
Fig. 8. Two-year postoperative follow-up panoramic radio-graph showing good ossification at the surgical site.
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expressed marginal sclerosis.5,8 Therefore, DF oftenmimics other common as well as unusual pathologiesof the jaws including ameloblastoma, odontogenicmyxoma, ABC, chondromyxoid fibroma, centralhemangioma, and eosinophilic granuloma.
The patient tolerated the surgical procedure well andwas discharged on postoperative day 3. Mild hypo-esthesia of the mental nerve was present for severaldays. She has been followed up for the past 2 yearswithout any clinical or radiographic signs of recurrence(Figure 8). Obvious ossification of the former resectioncavity was seen. The patient has shown excellentrecovery with remodeling of the inferior border and noneurosensory deficits.
DISCUSSIONDF has a relatively benign histologic appearance, but isthought to be belonging to a category between benignand malignant bone tumors because of its locallyaggressive nature. Therefore, some authors prefer the
term intraosseous desmoid tumor to DF. It representsthe osseous manifestation of aggressive fibromatosisand was first reported by Jaffe in 1958, who describedmultiple sites of involvement in the tibia, scapula, andfemur. Mandibular involvement is reported to beapproximately 22% of the various bony sites.5,6 Thefirst report of gnathic involvement with DF is attributedto Griffith and Irby in 1965, and since then numeroussimilar cases have been described.5,7-16
DFs most commonly occur in the metaphysis of thelong bones, the mandible, and the pelvis (approximately70% of occurrences), whereas the maxilla, calvaria,sternum, and vertebrae are less frequently affected. Itoccurs with equal frequency in both sexes and hasa predilection for patients in the first 3 decades of life.6
In the jaws, the posterior mandible is most frequentlyinvolved (the ramus, angle, and molar area). Thepremolar area and the anterior segments are lesscommonly affected.5,9 Our case was located in thepremolar region.6
Fibrous dysplasia can simulate DF in areas wherefibrous tissue predominates and osteoid production is
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not apparent. The distinction can be made by identi-fying areas of bone formation by additional sampling.Also the nuclei in fibrous dysplasia are shorter andmore compact-looking than the elongated, slendernuclei seen in DF. Nonossifying fibroma and solitarycongenital fibromatosis of bone can also be confusedwith DF.
Low-grade fibrosarcoma poses the most difficultproblem in the histologic differential diagnosis; in fact,the distinction may not always be possible and can onlybe detected when it recurs and metastasizes. However,fibrosarcoma is more cellular, with a recognizableherringbone pattern and plumper, larger cells than thosein DF. Cytologically, hyperchromasia with anaplasiaand mitotic activity quantitatively surpasses the raremitotic figures occasionally seen in DF.
Jaffe, in his discussion on the treatment of DF of bone,recommended segmental resection as the treatment ofchoice and noted that if the lesion is curetted and recurs,segmental resection or a more thorough curettage shouldbe performed. Wide resection or a thorough “marginal”curettage was the preferred method of treatment,whereas local or limited curettage often led to continuedgrowth of the tumor. It has been observed that curettageor peripheral ostectomy achieved with a bur drill ach-ieves better local tumor control.14,17
An important question that arises is whether thelesion removed 15 years ago is related to the currentlesion. Was the surgical approach too conservative inthe first treatment? Or are these 2 totally differentlesions and by coincidence were found almost in thesame region? Because of the lack of availability of theglass slide and block, we cannot answer that question.
In conclusion, the DF is a rare, well-differentiatedfibrous tumor with a slow but aggressive potential forgrowth. This lesion, although incapable of metasta-sizing, may recur locally when incompletely excised.Thorough curettage with possible extension of themargin with a bur (peripheral ostectomy) is the treat-ment of choice in early lesions. Lesions extendingbeyond the reactive bony rim require wide excision.14
The authors would like to thank Pr Georges Aftimos,Department of Pathology, Lebanese and St. Joseph Univer-sities; Director of National Institute of Pathology, for thehistology contribution and Pr Marcel Noujeim, DDS, MS,Department of Comprehensive Dentistry, Oral and Maxillo-facial Radiology Graduate Program Director, University of
Texas Health Science Center San Antonio, San Antonio,Texas, USA.
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Reprint requests:
Saydé Sokhn, CD, DUA, DUBSchool of DentistryLebanese UniversityBeirut, [email protected]
